To report a patient presenting with an intratarsal keratinous cyst of the Meibomian gland in the upper eyelid and a review of the relevant literature.

A 65-year-old male presented with a right upper eyelid mass which started 5 months prior. The patient reported that the mass recurred several weeks prior even after incision and curettage procedure. The mass was 9 x 5 mm in size and located in the center of the right upper eyelid at the level of lid crease, fixed to the tarsus and a whitish elevated focus was observed at the palpebral conjunctival surface. The mass was excised under local anesthesia and originated from the tarsus. The histopathological examinations revealed an intratarsal keratinous cyst composed of stratified squamous epithelium without keratohyalin granules and filled with keratin. The immunohistochemical studies showed positive staining results for cytokeratin 5/6, epithelial membrane antigen, and carcinoembryonic antigen.

Intratarsal keratinous cyst of the Meibomian gland should be considered as a differential diagnosis of a recurrent tarsal mass.